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Preliteracy impairments in children with neurofibromatosis type 1
Author(s) -
Arnold Shelley S,
Payne Jonathan M,
Lorenzo Jennifer,
North Kathryn N,
Barton Belinda
Publication year - 2018
Publication title -
developmental medicine and child neurology
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.658
H-Index - 143
eISSN - 1469-8749
pISSN - 0012-1622
DOI - 10.1111/dmcn.13768
Subject(s) - phonological awareness , spelling , psychology , neurofibromatosis , developmental psychology , audiology , cognition , phonological rule , reading (process) , literacy , phonology , clinical psychology , medicine , psychiatry , linguistics , pedagogy , philosophy , radiology
Aim This cross‐sectional study aimed to examine the preliteracy abilities of young children with neurofibromatosis type 1 ( NF 1) and to identify which of these abilities best predicted conventional literacy (spelling). Method Forty‐two children with NF 1 (23 males, 19 females; mean age [ SD ] 5y 6mo [6mo]) were compared with 32 unaffected children (15 males, 17 females; mean age [ SD ] 5y 4mo [6mo]). All children completed a comprehensive cognitive assessment including measures of phonological processing (phonological awareness, phonological memory, rapid automatic naming) and letter‐sound knowledge. Results Children with NF 1 performed significantly poorer than the comparison group across all cognitive and preliteracy domains, with specific weaknesses evident in phonological awareness ( F 1,68 =14.13, p <0.001, partial η 2 =0.17), phonological memory ( F 1,68 =13.87, p <0.001, partial η 2 =0.17), and letter‐sound knowledge ( F 1,71 =5.65, p =0.020, partial η 2 =0.07). Within the group with NF 1 group, over a third of children demonstrated impairment in at least one phonological processing domain and the risk of phonological impairment was 5.60 times that of unaffected children. Children's letter‐sound knowledge was the strongest predictor of conventional literacy (spelling). Interpretation This study establishes that preliteracy deficits are present and detectable in young children with NF 1. As a result of the high incidence of preliteracy impairment, we recommend screening phonological awareness and letter‐sound knowledge to identify risk of future learning disorders. What this paper adds Young children with neurofibromatosis type 1 are at elevated risk of preliteracy deficits. The most affected domains are phonological awareness and phonological memory. Letter‐sound knowledge is the strongest predictor of conventional literacy (spelling).